Hepatitis in Idiopathic Hypereosinophilic Syndrome: Report of an Unusual Case

Mahbuba Sharmin(1), Amin Lutful Kabir(2), Md. Abdul Aziz (3), Sonia Shormin Miah (4), Umme Shahera(5), Nahida Sultana (6), Khaza Amirul Islam(7)
(1) Bangabandhu Sheikh Mujib Medical University, Bangladesh
(2) Bangabandhu Sheikh Mujib Medical University, Bangladesh
(3) Bangabandhu Sheikh Mujib Medical University , Bangladesh
(4) Bangabandhu Sheikh Mujib Medical University, Bangladesh
(5) Bangabandhu Sheikh Mujib Medical University, Bangladesh
(6) Bangabandhu Sheikh Mujib Medical University, Bangladesh
(7) Shaheed Ziaur Rahman Medical College Hospital Bogura, Bangladesh

Abstract

World Health Organization defines a rare diagnosis Idiopathic hypereosinophilic syndrome (HES) as a persistent eosinophilia for 6 months and resulting in end-organ dysfunction. Most of the patients present with nonspecific symptoms, while others will present with symptoms of the affected organs, commonly those involving the heart, skin, or nervous system. Gastrointestinal or liver involvement is estimated to affect up to one-third of patients with HES, although patients with clinically significant disease are limited to case reports. This is the first report of a patient presenting with idiopathic HES related hepatitis and achalasia. Hypereosinophilic syndrome has been reported to be associated with hepatic dysfunction; liver histology is mainly characterized by a diffuse eosinophilic inflammatory infiltrate. A 49-yr-old woman, diagnosed as a case of idiopathic hypereosinophilic syndrome with bone marrow and pulmonary eosinophilic infiltrates associated with peripheral eosinophilia,high IgE level  developed features of chronic gatrornteritis, hepatitis, with a significant eosinophil component. She responded well to systemic glucocorticoid  and Imatinib therapy with normalization of liver function tests within a few weeks.

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References

Klion AD, Bochner BS, Gleich GJ, Nutman TB, Rothenberg ME, Simon HU, Wechsler ME, Weller PF, The Hypereosinophilic Syndromes Working Group. Approaches to the treatment of hypereosinophilic syndromes. J Allergy Clin Immunol. 2006 Jun;117(6):1292-302. doi: 10.1016/j.jaci.2006.02.042.

Klion AD. How I treat hypereosinophilic syndromes. Blood. 2009 Oct 29; 114(18): 3736–3741.

Simon HU, Rothenberg ME, Bochner BS, Weller PF, Wardlaw AJ, Wechsler ME, Rosenwasser LJ, Roufosse F, Gleich GJ, Klion AD. Refining the definition of hypereosinophilic syndrome. J Allergy Clin Immunol. 2010 Jul; 126(1):45-9. doi: 10.1016/j.jaci.2010.03.042.

Chusid MJ, Dale DC, West BC, Wolff SM. The hypereosinophilic syndrome: analysis of fourteen cases with review of the literature. Medicine (Baltimore). 1975 Jan; 54(1):1-27.

Klion AD (2011) Eosinophilic myeloproliferative disorders. Hematology Am Soc Hematol Educ Program. 2011;2011:257-63. doi: 10.1182/asheducation-2011.1.257.

Ogbogu PU, Bochner BS, Butterfield JH, Gleich GJ, Huss-Marp J, Kahn JE, Leiferman KM, Nutman TB, Pfab F, Ring J, Rothenberg ME, Roufosse F, Sajous MH, Sheikh J, Simon D, Simon HU, Stein ML, Wardlaw A, Weller PF, Klion AD. Hypereosinophilic syndrome: a multicenter, retrospective analysis of clinical characteristics and response to therapy. J Allergy Clin Immunol. 2009 Dec;124(6):1319-25.e3. doi: 10.1016/j.jaci.2009.09.022.

Roufosse FE, Goldman M, Cogan E. Hypereosinophilic syndromes. Orphanet J Rare Dis. 2007 Sep 11;2:37. doi: 10.1186/1750-1172-2-37.

Weller PF, Bubley GJ. The idiopathic hypereosinophilic syndrome. Blood. 1994 May 15;83(10):2759-79.

Nallegowda M, Sing U, Handa G, Arora B. Idiopathic Hypereosinophilic Syndrome with Stroke in Young: A Case Report. IJPMR. 2003; 14: 24-26.

Weingarten JS, O’sheal SF, Margolis WS. Eosinophilic meningitis and the HES: case report and review of the literature. Am J Med 198578: 674-676.

Choi HY, Park HJ, Won YS, Jung JW, Park S. A Case of Eosinophilic Meningitis Associated with Idiopathic Hypereosinophilic Syndrome. J Korean Neurol Assoc 200523: 396-398.

Hardy WR, Anderson RE. The hypereosinophilic syndrome. Ann Intern Med 196868: 1220-1229.

Venkatesh C, Mahender E, Janani S, Malathi S, Vijayakumar M, Nammalwar BR. Hypereosinophilic Syndrome. Indian J Peadiatrics 200673: 237-239.

Authors

Mahbuba Sharmin
dr.sharmin1012@gmail.com (Primary Contact)
Amin Lutful Kabir
Md. Abdul Aziz
Sonia Shormin Miah
Umme Shahera
Nahida Sultana
Khaza Amirul Islam
Author Biographies

Mahbuba Sharmin, Bangabandhu Sheikh Mujib Medical University

Medical Officer, Bangabandhu Sheikh Mujib Medical University

Amin Lutful Kabir, Bangabandhu Sheikh Mujib Medical University

Associate Professor of Haematology, Bangabandhu Sheikh Mujib Medical University

Umme Shahera, Bangabandhu Sheikh Mujib Medical University

Assistant Professor, Department of Virology, Bangabandhu Sheikh Mujib Medical University

Nahida Sultana , Bangabandhu Sheikh Mujib Medical University

Research Assistant, Bangabandhu Sheikh Mujib Medical University

Khaza Amirul Islam, Shaheed Ziaur Rahman Medical College Hospital Bogura

Medical Officer, Shaheed Ziaur Rahman Medical College Hospital Bogura

1.
Sharmin M, Kabir AL, Aziz MA, Miah SS, Shahera U, Sultana N, et al. Hepatitis in Idiopathic Hypereosinophilic Syndrome: Report of an Unusual Case. Haematol J Bangladesh [Internet]. 2021 Nov. 30 [cited 2024 Jun. 10];5(2):69-72. Available from: https://journal.hematologybd.org/index.php/haematoljbd/article/view/73

Article Details

How to Cite

1.
Sharmin M, Kabir AL, Aziz MA, Miah SS, Shahera U, Sultana N, et al. Hepatitis in Idiopathic Hypereosinophilic Syndrome: Report of an Unusual Case. Haematol J Bangladesh [Internet]. 2021 Nov. 30 [cited 2024 Jun. 10];5(2):69-72. Available from: https://journal.hematologybd.org/index.php/haematoljbd/article/view/73
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